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die. After their honeymoon, Hans is reunited with his first love
Princess Bertha. Ondine leaves Hans in an attempt to protect him, but she is recaptured by a fisherman and Hans is stricken by the King's curse. On meeting Ondine again, Hans tells her that "all the things my body once did by itself, it does now only by special order ... A single moment of inattention and I forget to breathe". Hans and Ondine kiss, after which he dies.
429:
169:(difficulty swallowing) and anomalies of the pupilla. Other symptoms include darkening of skin color from inadequate amounts of oxygen, drowsiness, fatigue, headaches, and an inability to sleep at night. Patients with CHS also have a sensitivity to sedatives and narcotics, which makes respiration even more difficult. A low concentration of oxygen in the
408:
to earlier
European folk tales. The water-spirit Ondine tells her future husband Hans, whom she had just met, that "I shall be the shoes of your feet ... I shall be the breath of your lungs". Ondine reluctantly makes a pact with her uncle the King of the Ondines that if Hans ever deceives her he will
268:
because part of the autonomic nervous system (such as sympathetic ganglia) derives from the neural crest. However, this denomination is no longer favored because essential neurons of the autonomic nervous system, including those that underlie the defining symptom of the disease (respiratory arrests),
140:
Although rare, cases of long-term untreated CCHS have been reported and are termed late onset CCHS (LO-CCHS). There have, however, even been cases of LO-CCHS where family members found to have it have been asymptomatic. Again, lack of awareness in the medical community may cause such a delay. CCHS
132:
of breathing. In 2006, there were only about 200 known cases worldwide. As of 2008, only 1000 total cases were known. The diagnosis may be delayed because of variations in the severity of the manifestations or lack of awareness in the medical community, particularly in milder cases. However, as
329:
Physicians unable to recognize the disorder should seek help from a neurologist and a pulmonologist. In some locations, such as France, optimal management of patients, once identified, has been aided by the creation of a national registry and the formation of a network of centers.
342:
can effectively be used without the need for a tracheotomy. Other potential treatments for CCHS include oxygen therapy and medicine for stimulating the respiratory system. Currently, problems arise with the extended use of ventilators, including fatal infections and pneumonia.
177:
or a failure of the right side of the heart. Associated complications may also include gastro-esophageal reflux, ophthalmologic issues, seizures, recurrent pneumonia, developmental delays, learning disabilities, episodes of fainting, and temperature disregulation.
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and its connection to the diagnosis. Most frequently, Ondine was inaccurately blamed for cursing Hans, but other mistakes sometimes changed the nature of the curse itself. Such errors led to confusion in defining the medical condition.
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there have been cases where asymptomatic family members also were found to have CCHS, it may be that these figures only reflect those found to require mechanical ventilation. In all cases, episodes of
530:
Windisch W, Hennings E, Storre J, Matthys H, Sorichter S (2004). "Long-term survival of a patient with congenital central hypoventilation syndrome despite the lack of continuous ventilatory support".
114:
during sleep (and during wakefulness in severe cases). CHS can either be congenital (CCHS) or acquired (ACHS) later in life. The condition can be fatal if untreated. CCHS was once known as
362:
Most people with CCHS (unless they have the Late Onset form) do not survive infancy, unless they receive ventilatory assistance during sleep. An alternative to a mechanical ventilator is
670:
Trang H, Dehan M, Beaufils F, Zaccaria I, Amiel J, Gaultier C (2005). "The French
Congenital Central Hypoventilation Syndrome Registry: general data, phenotype, and genotype".
311:
is present during other nonrapid eye movement sleep stages and even wakefulness. A subset of CCHS patients are at very high risk for developing malignant neural crest-derived
289:, usually in the first months of life. Medical evaluation excludes lesions of the brain, heart, and lungs but demonstrates impaired responses to build-up of carbon dioxide (
17:
1147:
Takeda, S.; Fujii, Y.; Kawahara, H.; Nakahara, K.; Matsuda H. (1996). "Central alveolar hypoventilation syndrome (Undine's curse) with gastroesophageal reflux".
48:
Ondine's curse, primary alveolar hypoventilation, alveolar hypoventilation secondary to neurologic disease, idiopathic acquired central hypoventilation syndrome
245:
Medical investigation of patients with this syndrome has led to a deeper understanding of how the body and brain regulate breathing on a molecular level.
889:"Facial phenotype in children and young adults with PHOX2B-determined congenital central hypoventilation syndrome: quantitative pattern of dysmorphology"
338:
People generally require tracheostomy and lifetime mechanical ventilation on a ventilator in order to survive. However, it has now been shown that
1074:"Congenital central hypoventilation syndrome: An overview of etiopathogenesis, associated pathologies, clinical presentation, and management"
624:
Lovell BL, Bullock RE, Anderson KN (March 2010). "An unusual presentation of congenital central hypoventilation syndrome (Ondine's Curse)".
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Since being coined in 1962 the name has become controversial in medical literature, as later summaries frequently misunderstood the plot of
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1127:
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CCHS was first described in 1962 by
Severinghaus and Mitchell in three patients following surgery to the upper cervical
1002:
Kincaid PK, Dietrich RB, Pais MJ (1994). "Pediatric case of the day. Neurocristopathy (Ondine-Hirschsprung syndrome)".
1182:
Severinghaus J.W.; Mitchell, R. A. (1962). "Undine's curse — failure of respiratory center automaticity while awake".
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disorder, but in rare circumstances, can also result from severe brain or spinal trauma or injury (such as after an
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Long, K. J.; Allen, N. (October 1984). "Abnormal brain-stem auditory evoked potentials following Ondine's curse".
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277:), not from the neural crest, which may be the case in other mixed embryological origins and neurocristopathies.
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Poceta, J. S.; Strandjord, T. P.; Badura, R. J.; Milstein, J. M. (1987). "Undine curse and neurocristopathy".
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occur in sleep, but in a few patients, at the most severe end of the spectrum, apnea also occurs while awake.
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1970:
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249:, a transcription factor involved in the development of neurons, can be associated with this condition. This
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354:, a US based research charity which focuses the vast majority of its funding on CCHS scientific research.
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234:. Long and Allen (1984) were the first to report the abnormal brainstem auditory-evoked responses in an
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also may cause hypoxia-induced pulmonary vasoconstriction and pulmonary hypertension, culminating in
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575:"Extreme intra-familial variability of congenital central hypoventilation syndrome: a case series"
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woman who recovered from Ondine's curse. These investigators hypothesized that their patient's
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There are two primary organisations focused on delivering new treatments for CCHS. These are
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Zaidi, Saher; Gandhi, Jason; Vatsia, Sohrab; Smith, Noel L.; Khan, Sardar Ali (March 2018).
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447:, an Oscar-nominated 2013 short documentary film about a child with Ondine's curse
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CHS is associated with respiratory arrests during sleep and, in some cases, to
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955:"Enteric nervous system development: migration, differentiation, and disease"
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Bygarski, Elizabeth; Paterson, Melanie; Lemire, Edmond G. (April 26, 2013).
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848:"Pediatric disorders with autonomic dysfunction: what role for PHOX2B?"
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Its name is a reference to the story of Ondine and Hans, characters in
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2005:
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American
Journal of Physiology. Gastrointestinal and Liver Physiology
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Children with CCHS develop life-threatening episodes of apnea with
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2020:
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350:, a UK based research charity which focuses solely on CCHS and
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revealed mutations in 91% of the cases within a French cohort.
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ACHS can develop as a result of severe injury or trauma to the
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Todd ES, Weinberg SM, Berry-Kravis EM, et al. (2006).
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susceptibility is not known to be affected by sex or race.
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128:. Congenital cases are very rare and involve a failure of
2000:
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804:
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Jazeela Fayyaz, DO (2017-12-05). Zab
Mosenifar, M (ed.).
297:), the two strongest stimuli to increase breathing rate.
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712:. U.S. National Library of Medicine. September 17, 2010.
1071:
807:"Genetics and early disturbances of breathing control"
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was poisoned—not destroyed—by her chronic alcoholism.
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1201:
Sugar, Oscar (1978). "In Search of Ondine's Curse".
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424:
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805:Gaultier C; Amiel J; Dauger S; et al. (2004).
706:"Primary alveolar hypoventilation: Ondine's curse"
665:
663:
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846:Gaultier C, Trang H, Dauger S, Gallego J (2005).
724:"CCHS Network | Casting a Light in the Dark"
303:shows that hypoventilation is most marked during
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953:Lake, Jonathan; Heuckeroth, Robert (July 2013).
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253:is important for the normal development of the
1128:"Death by Nap: Boy Risks Death if He Nods off"
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293:) and decreases of oxygen in the circulation (
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485:"Congenital central hypoventilation syndrome"
1355:Congenital central hypoventilation syndrome
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514:Congenital Central Hypoventilation Syndrome
1704:Rapid eye movement sleep behavior disorder
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401:based on traditions tracing back through
18:Central alveolar hypoventilation syndrome
784:(18th ed.). New York: McGraw-Hill.
260:The disease used to be classified as a "
778:"Chapter 264. Disorders of Ventilation"
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755:10.1001/archneur.1984.04050210111028
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491:. U.S. National Library of Medicine
106:that causes ineffective breathing,
24:
1016:10.1148/radiographics.14.5.7991820
906:10.1203/01.pdr.0000191814.73340.1d
865:10.1203/01.PDR.0000166755.29277.C4
824:10.1203/01.PDR.0000115677.78759.C5
25:
2112:
1229:
929:"Gene secret of 'mythical curse'"
1544:Obesity hypoventilation syndrome
1539:Central hypoventilation syndrome
1211:10.1001/jama.1978.03290030054019
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186:CHS is exhibited typically as a
104:sleep-related breathing disorder
96:Central hypoventilation syndrome
40:Central hypoventilation syndrome
1699:Periodic limb movement disorder
1666:Non-24-hour sleep–wake disorder
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1971:Biphasic and polyphasic sleep
1779:Nocturnal clitoral tumescence
1641:Advanced sleep phase disorder
710:A.D.A.M. Medical Encyclopedia
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1651:Delayed sleep phase disorder
1579:Excessive daytime sleepiness
1090:10.1016/j.autneu.2017.11.003
1051:10.1016/0887-8994(87)90011-7
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340:biphasic cuirass ventilation
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307:. In the most severe cases,
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7:
1784:Nocturnal penile tumescence
1656:Irregular sleep–wake rhythm
467:"Hypoventilation Syndromes"
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10:
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1646:Cyclic alternating pattern
1205:. 1978, 240 (3): 236–237.
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1862:Behavioral sleep medicine
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161:(partial agenesis of the
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1419:Rapid eye movement (REM)
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255:autonomic nervous system
27:Not to be confused with
2081:Breathing abnormalities
1740:Exploding head syndrome
1549:Obstructive sleep apnea
1161:10.1378/chest.110.3.850
939:(The article misspells
638:10.1136/emj.2009.072215
592:10.1186/1752-1947-7-117
489:Genetics Home Reference
322:Sequencing of the gene
228:multiple system atrophy
218:) or due to particular
214:, as a complication of
67:John William Waterhouse
2091:Neurological disorders
2055:Sleeping while on duty
1604:Idiopathic hypersomnia
1078:Autonomic Neuroscience
776:Longo, Dan L. (2012).
684:10.1378/chest.127.1.72
163:enteric nervous system
1877:Neuroscience of sleep
1609:Night eating syndrome
1594:Kleine–Levin syndrome
269:are derived from the
264:", or disease of the
2086:Respiratory diseases
2031:Sleep and creativity
159:Hirschsprung disease
2026:Sleep and breathing
1482:Sensorimotor rhythm
406:(a novella of 1811)
224:Parkinson's disease
222:conditions such as
192:automobile accident
155:sympathetic ganglia
29:Central sleep apnea
2036:Sleep and learning
1789:Nocturnal emission
1689:Nightmare disorder
1554:Periodic breathing
1319:External resources
232:multiple sclerosis
145:Signs and symptoms
112:respiratory arrest
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544:10.1159/000076685
348:Keep Me Breathing
220:neurodegenerative
130:autonomic control
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34:Medical condition
16:(Redirected from
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1981:Circadian rhythm
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352:The CCHS Network
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153:(tumors of the
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1813:
1812:Sleep hygiene
1810:
1808:
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1800:
1796:
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1770:
1769:Sleep inertia
1767:
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1748:
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1736:
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1719:Sleep-talking
1717:
1715:
1714:Sleep driving
1712:
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1697:
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1487:Sleep spindle
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1136:. 2010-08-17.
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1009:
1005:
1004:Radiographics
998:
990:
986:
981:
976:
972:
968:
965:(1): G1–G24.
964:
960:
956:
949:
943:as "Thox2b".)
942:
937:. 5 May 2003.
936:
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916:
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883:
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834:
830:
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820:
817:(5): 729–33.
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791:9780071748896
787:
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772:
764:
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331:
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317:neuroblastoma
314:
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251:homeobox gene
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237:
233:
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201:
197:
193:
189:
179:
176:
175:cor pulmonale
172:
168:
164:
160:
156:
152:
151:neuroblastoma
142:
138:
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131:
127:
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118:
117:
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109:
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101:
97:
87:
81:
78:
76:
72:
68:
64:
60:
56:
51:
47:
43:
38:
30:
19:
1932:Sleeping bag
1709:Sleepwalking
1694:Night terror
1538:
1411:sleep cycles
1348:
1337:
1326:
1302:
1291:
1280:
1265:
1250:
1202:
1196:
1187:
1183:
1177:
1152:
1148:
1142:
1131:
1122:
1081:
1077:
1067:
1045:(6): 370–2.
1042:
1038:
1032:
1007:
1003:
997:
962:
958:
948:
932:
923:
899:(1): 39–45.
896:
893:Pediatr. Res
892:
882:
855:
852:Pediatr. Res
851:
841:
814:
811:Pediatr. Res
810:
800:
781:
771:
746:
743:Arch. Neurol
742:
736:
728:CCHS Network
727:
718:
709:
700:
675:
671:
632:(3): 237–8.
629:
625:
619:
582:
578:
568:
538:(2): 195–8.
535:
531:
525:
512:
493:. Retrieved
488:
479:
470:
460:
442:
413:
411:
403:
392:
389:
373:
361:
345:
337:
328:
321:
299:
284:
266:neural crest
259:
244:
216:neurosurgery
208:encephalitis
200:asphyxiation
185:
148:
139:
119:
115:
99:
95:
94:
62:
2016:Second wind
1991:Dream diary
1867:Sleep study
1807:Sleep diary
1759:Hypnopompia
1754:Sleep onset
1745:Hypnic jerk
1584:Hypersomnia
1534:Catathrenia
1529:Sleep apnea
1439:Brain waves
1409:Stages of
1350:GeneReviews
1328:MedlinePlus
678:(1): 72–9.
626:Emerg Med J
532:Respiration
376:spinal cord
291:hypercapnia
271:neural tube
204:brain tumor
69:(1849–1917)
45:Other names
2075:Categories
2050:Sleep debt
1996:Microsleep
1976:Chronotype
1890:Daily life
1774:Somnolence
1750:Hypnagogia
1681:Parasomnia
1599:Narcolepsy
1511:Anatomical
1492:Theta wave
1462:Gamma wave
1457:Delta wave
1447:Alpha wave
1304:DiseasesDB
858:(1): 1–6.
585:(1): 117.
452:References
315:, such as
188:congenital
2060:Sleepover
2011:Power nap
2006:Nightwear
1872:Melatonin
1834:Somnology
1799:Treatment
1632:disorders
1571:Dyssomnia
1477:PGO waves
1472:Mu rhythm
1467:K-complex
1452:Beta wave
1429:Slow-wave
1339:eMedicine
1098:1566-0702
654:206938512
519:eMedicine
444:Our Curse
386:Etymology
380:brainstem
358:Prognosis
334:Treatment
281:Diagnosis
240:brainstem
236:alcoholic
212:poisoning
167:dysphagia
126:brainstem
80:Neurology
75:Specialty
1927:Mattress
1902:Bunk bed
1822:Hypnosis
1614:Nocturia
1589:Insomnia
1184:Clin Res
1133:ABC News
1106:29249648
989:23639815
934:BBC News
915:16327002
874:15901893
833:14739359
692:15653965
646:20304901
611:23622117
560:36554567
552:15031579
471:Medscape
421:See also
287:cyanosis
1954:Bedtime
1949:Bedroom
1944:Bedding
1939:Bed bug
1922:Hammock
1827:Lullaby
1661:Jet lag
1561:Snoring
1519:Bruxism
1298:D020182
1169:8797441
1114:4435638
1084:: 1–9.
1059:3508086
1024:7991820
980:3725693
763:6477223
602:3651317
370:History
295:hypoxia
275:medulla
102:) is a
2021:Siesta
1907:Daybed
1735:Dreams
1333:000078
1287:209880
1276:327.25
1261:G47.35
1219:351225
1217:
1190:: 122.
1167:
1112:
1104:
1096:
1057:
1022:
987:
977:
941:PHOX2B
913:
872:
831:
788:
761:
690:
652:
644:
609:
599:
558:
550:
495:5 June
414:Ondine
404:Undine
393:Ondine
324:PHOX2B
313:tumors
247:PHOX2B
196:stroke
182:Causes
82:
63:Ondine
1964:Story
1917:Futon
1850:Other
1398:Sleep
1309:32976
1149:Chest
1110:S2CID
672:Chest
650:S2CID
556:S2CID
273:(the
230:, or
135:apnea
122:brain
110:, or
108:apnea
1400:and
1293:MeSH
1282:OMIM
1271:9-CM
1215:PMID
1203:JAMA
1165:PMID
1102:PMID
1094:ISSN
1055:PMID
1020:PMID
985:PMID
911:PMID
870:PMID
829:PMID
786:ISBN
759:PMID
688:PMID
642:PMID
607:PMID
548:PMID
497:2015
378:and
2001:Nap
1897:Bed
1267:ICD
1252:ICD
1207:doi
1157:doi
1153:110
1086:doi
1082:210
1047:doi
1012:doi
975:PMC
967:doi
963:305
901:doi
860:doi
819:doi
751:doi
680:doi
676:127
634:doi
597:PMC
587:doi
540:doi
517:at
397:by
165:),
157:),
124:or
100:CHS
65:by
2077::
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1342::
1331::
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1274::
1259::
1256:10
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1188:10
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1151:.
1130:.
1108:.
1100:.
1092:.
1080:.
1076:.
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1041:.
1018:.
1008:14
1006:.
983:.
973:.
961:.
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931:.
909:.
897:59
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856:58
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850:.
827:.
815:55
813:.
809:.
780:.
757:.
747:41
745:.
726:.
708:.
686:.
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662:^
648:.
640:.
630:27
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595:.
581:.
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505:^
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1014::
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903::
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